Adenoid cystic carcinoma of the Bartholin gland is not HPV-related: A case report and review of literature

Pathol Res Pract. 2020 Jun;216(6):152968. doi: 10.1016/j.prp.2020.152968. Epub 2020 Apr 19.

Abstract

Adenoid cystic carcinoma (ACC) of the Bartholin gland is a rare gynaecological entity. Despite its slow growth and inconspicuous presentation, vulvar ACC has a propensity for perineural invasion and is therefore associated with high local recurrence rates. We report a case of vulvar ACC in a 61-year-old woman with a prolonged swelling of the Bartholin gland. This patient presented with pulmonary metastases at the moment of histological diagnosis. The vulvar and the pulmonary lesions showed identical histology. Despite a history of human papilloma virus (HPV)-related usual type vulvar intra-epithelial neoplasia and cervical squamous cell carcinoma, the vulvar ACC was negative for both p16 immunohistochemistry and HPV-DNA. We conclude that HPV is not involved in the pathogenesis of pure ACC of the Bartholin gland. Additionally, we advocate a low threshold for performing biopsies of vulvar swellings in women aged >40 years, to rule out malignancy and to prevent diagnostic delays.

Keywords: Adenoid cystic carcinoma; Bartholin gland; Human papilloma virus; Lung metastasis; Vulva; p16.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Bartholin's Glands / pathology*
  • Carcinoma, Adenoid Cystic / pathology*
  • Carcinoma, Squamous Cell / pathology
  • Carcinoma, Squamous Cell / virology
  • Female
  • Humans
  • Lung Neoplasms / secondary
  • Middle Aged
  • Neoplasms, Second Primary / pathology*
  • Papillomavirus Infections / complications
  • Uterine Cervical Neoplasms / pathology
  • Uterine Cervical Neoplasms / virology
  • Vulvar Neoplasms / pathology*